Kulozik A E, Kar B C, Satapathy R K, Serjeant B E, Serjeant G R, Weatherall D J
Blood. 1987 Jun;69(6):1742-6.
To further explore the cause for variation in hemoglobin F (Hb F) levels in sickle cell disease, the beta globin restriction-fragment length polymorphism haplotypes were determined in a total of 303 (126 SS, 141 AS, 17 S beta(0), 7 A beta, (0) and 12 AA) Indians from the state of Orissa. The beta(s) globin gene was found to be linked almost exclusively to a beta(S) haplotype ( -++-), which is also common in Saudi Arabian patients from the Eastern Province (referred to as the Asian beta(s) haplotype). By contrast, the majority of beta A and beta(0) thalassemia globin genes are linked to haplotypes common in all European and Asian populations (+-----[+/-]; --++-++). Family studies showed that there is a genetic factor elevating Hb F levels dominantly in homozygotes (SS). This factor appears to be related to the Asian beta(s) globin haplotype, and a mechanism for its action is discussed. There is also a high prevalence of an independent Swiss type hereditary persistence of fetal hemoglobin (HPFH) determinant active in both the sickle cell trait and in sickle cell disease.
为进一步探究镰状细胞病患者血红蛋白F(Hb F)水平存在差异的原因,对来自奥里萨邦的总共303名印度人(126名SS型、141名AS型、17名Sβ(0)型、7名Aβ(0)型和12名AA型)的β珠蛋白限制性片段长度多态性单倍型进行了测定。发现β(S)珠蛋白基因几乎仅与一种β(S)单倍型(-++-)连锁,这种单倍型在沙特阿拉伯东部省份的患者中也很常见(称为亚洲β(S)单倍型)。相比之下,大多数βA和β(0)地中海贫血珠蛋白基因与所有欧洲和亚洲人群中常见的单倍型连锁(+-----[+/-];--++-++)。家系研究表明,存在一个在纯合子(SS)中显性升高Hb F水平的遗传因素。该因素似乎与亚洲β(S)珠蛋白单倍型有关,并对其作用机制进行了讨论。在镰状细胞性状和镰状细胞病中均活跃的一种独立的瑞士型胎儿血红蛋白遗传性持续存在(HPFH)决定因素也具有较高的发生率。